A ZFHX4 mutation associated with a recognizable neuropsychological and facial phenotype.


Journal

European journal of medical genetics
ISSN: 1878-0849
Titre abrégé: Eur J Med Genet
Pays: Netherlands
ID NLM: 101247089

Informations de publication

Date de publication:
Nov 2021
Historique:
received: 13 03 2021
revised: 27 07 2021
accepted: 22 08 2021
pubmed: 31 8 2021
medline: 13 1 2022
entrez: 30 8 2021
Statut: ppublish

Résumé

Several patients with chromosomal deletions including ZFHX4 gene have been described, whereas point mutations are very rare. This gene encodes for a transcription factor involved in the development of several embryonal processes, including brain differentiation. Patients with 8q21.11 deletions usually show intellectual disability, short stature, peculiar facial features, and severe eye abnormalities. We describe a female patient with mild intellectual disability, autism spectrum disorder, strabismus, ptosis, low-set and prominent ears, high-arched palate, microretrognathia. Clinical Exome Sequencing revealed the presence of a de novo heterozygous variant in ZFHX4. Therefore, we further investigate the different phenotypes of ZFHX4 mutations and 8q21.11 deletions.

Identifiants

pubmed: 34461323
pii: S1769-7212(21)00187-7
doi: 10.1016/j.ejmg.2021.104321
pii:
doi:

Substances chimiques

Homeodomain Proteins 0
Transcription Factors 0
ZFHX4 protein, human 0

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

104321

Informations de copyright

Copyright © 2021 Elsevier Masson SAS. All rights reserved.

Auteurs

Paolo Fontana (P)

Medical Genetics Unit - P.O. Gaetano Rummo - A.O.R.N. San Pio, Benevento, BN, Italy. Electronic address: paolo.fontana@ao-rummo.it.

Monia Ginevrino (M)

Translational Cytogenomics Research Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy; Università Cattolica del Sacro Cuore, Fondazione Policlinico Universitario "A. Gemelli" IRCCS, Roma, Italy.

Kristel Bejo (K)

Translational Cytogenomics Research Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.

Giuseppina Cantalupo (G)

Medical Genetics Unit - P.O. Gaetano Rummo - A.O.R.N. San Pio, Benevento, BN, Italy.

Maria Ciavarella (M)

Medical Genetics Unit - P.O. Gaetano Rummo - A.O.R.N. San Pio, Benevento, BN, Italy.

Cinzia Lombardi (C)

Medical Genetics Unit - P.O. Gaetano Rummo - A.O.R.N. San Pio, Benevento, BN, Italy.

Marianna Maioli (M)

Medical Genetics Unit - P.O. Gaetano Rummo - A.O.R.N. San Pio, Benevento, BN, Italy.

Francesca Scarano (F)

Medical Genetics Unit - P.O. Gaetano Rummo - A.O.R.N. San Pio, Benevento, BN, Italy.

Claudia Costabile (C)

Medical Genetics Unit - P.O. Gaetano Rummo - A.O.R.N. San Pio, Benevento, BN, Italy.

Antonio Novelli (A)

Translational Cytogenomics Research Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.

Fortunato Lonardo (F)

Medical Genetics Unit - P.O. Gaetano Rummo - A.O.R.N. San Pio, Benevento, BN, Italy.

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Classifications MeSH